Factitious triplegia: Case report

Factitious triplegia: Case report

1161 CLINICAL NOTES Factitious Triplegia: Case Report John F. Berry, MD, Rodney E. Hillis, MS, Susan E. Hi&man, MSW ABSTRACT. Berry JF, Hillis RE...

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1161

CLINICAL NOTES

Factitious Triplegia: Case Report John F. Berry, MD, Rodney

E. Hillis, MS, Susan E. Hi&man,

MSW

ABSTRACT. Berry JF, Hillis RE, Hitxman SE. Factitious Triplegia: case report. Arch Phys Med Rehabil 1994;75:1161-4. l A 37-year-old woman was admitted for rehabilitation of triplegia with diagnoses of conversion disorder and possible multiple sclerosis. Attempts to manage the patient with techniques often effective with conversion disorders were unsuccessful, and the patient was discharged to a nursing home. After the development of further symptoms and a second extensive medical work-up, the factitious nature of her symptoms became evident. Awareness of the characteristics of factitious disorders, early background investigation, and psychiatric consuhation can facilitate the appropriate management and referral of these patients, and spare rehabilitation providers the frustrations and possible medicolegal complications of patients with this disorder. 0 1994 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation

Individuals with factitious disorders may present with acute or subacute neurological deficits. Because they are often medically sophisticated, determining that their symptoms are not caused by actual physical pathology can be difficult; potentially even more difficult is discriminating factitious disorders from conversion disorders and malingering. Conversion disorders, factitious disorders, and malingering are felt by some authorsle4 to represent a spectrum in which symptoms and objective findings may be similar, but the individual’s conscious awareness and control over symptoms varies. Although successful rehabilitation management of individuals with conversion disorder has been described,5 treatment of factitious disorders is controversial and of questionable efficacy.2*6 Several cases of successful treatment of chronic factitious disorder with physical symptoms (Munchausen syndrome) have been reported; however, in one instance this required 3 years of psychiatric hospitalization,7 and in the second aversive conditioning was used.8 Current cost and medicolegal concerns limit the applicability of either approach. Although behavior modification techniques have been effective in many areas of rehabilitation, this effectiveness may not extend to the treatment of disability secondary to Munchausen syndrome, as the following case illustrates.

Case A 37-year-old woman with a stated history of recent deep vein thrombosis and pulmonary embolism presented on two occasions to the emergency room with complaints of blurred vision, lightheadedness, and fall with injury to the head and shoulder. On the second occasion, she also complained of chest pain, cough, fever, From the Section of Physical Medicine and Rehabilitation, Louisiana State Universitv Medical Center (Dr. Berrv. Mr. Hillis), and Deuartment of Social Services, Louisiana Rehabilitatibn Institakz (Ms. Hitznk), New Orleans, LA. Submitted for publication March 30, 1994. Accepted in revised form June 2, 1994. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the authors or upon any organizations with which the authors are associated. Reprint requests to John F. Berry, MD, Section of Physical Medicine and Rehabilitation, Louisiana State University Medical Center, 1542 Tulane Avenue, New Orleans, LA 70112. 0 1994 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation 0003-9993/94/7510-3031$3.00/O

and general malaise. Physical examination was unremarkable except for orthostatic hypotension, a few crackles at the lung bases, mild weakness in the left leg, and reduced vibratory sensation over both feet. She was admitted to rule out recurrent pulmonary embolism and for work-up of syncope and upper respiratory infection. Initial screening laboratory studies were unremarkable. Heparin anticoagulation was initiated but discontinued when ventilationperfusion scanning showed low probability of pulmonary embolism. She continued to complain of episodic severe chest pain, which was relieved by nitroglycerin. Neurological consultation was obtained for work-up of syncope. On her fourth hospital day, she complained of sudden onset of severe headache, and was found on examination to have new dysarthria, right hemiparesis, and right hemianesthesia. Emergency computed tomography (CT) scan was normal. During the next several days her symptoms progressed to bilateral lower extremity and right upper extremity paralysis and urinary and fecal retention. The neurology consultant felt that her symptoms were probably functional, but that an underlying demyelinating disease was possible; magnetic resonance imaging, somatosensory evoked responses, and brainstem auditory evoked responses were normal, but nondiagnostic abnormalities were noted on visual evoked response studies (VERS). Psychiatry consultation was obtained. Although conversion disorder or factitious disorder were considered as possibilities, the consultant felt that underlying physical pathology was probable, and that the patient should have a complete medical work-up. Subsequently consultations were obtained from cardiology, urology, and rheumatology; extensive studies were negative except for mildly elevated erythrocyte sedimentation rate (ESR) on two occasions, and an antinuclear antibody (ANA) titer of 1:40 speckled. Physical medicine consultation was obtained, and the patient was transferred to the rehabilitation unit on her 26th hospital day with the admission diagnoses of triplegia secondary to conversion disorder and possible multiple sclerosis. Admission physical examination showed a mildly overweight, apparently depressed woman with inability to voluntarily move her legs or right arm. Both legs were held in rigid extension with ankles plantar flexed; during the formal examination these groups could not be broken by the examiner, but the patient was later able to relax her legs for transfers and catheterization. The right arm was flaccid at the shoulder and elbow, but the right hand was held tightly fisted; when strong extension force was applied, the digits became tremulous, then collapsed into flaccid extension. Sensation to light touch and pin was absent over the three affected extremities, with no response Arch Phys Mad Rehebil Vol75, October 1994

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elicited to strong stimulation with pin even when applied unobserved by the patient. Deep tendon reflexes were normal in the arms; lower extremity deep tendon and plantar reflexes could not be assessed because of her rigidity. The remainder of the physical examination was normal. A behavioral treatment program similar to those previously described for conversion disorder? was developed by the treatment team and agreed to by the patient. The program began with the patient gradually brought to the standing position in a tilt table, with planned progression to standing in the parallel bars, weight shifting, stepping in place, and assisted gait in the parallel bars. Because she held her legs in rigid extension during all examinations, the team felt that standing would be easily achieved. However, when elevated to the standing position, the patient’s legs consistently became flaccid, and she had to be supported by the therapist and the tilt-table straps. Increasing the gradualness of the approach and provision of ample praise for small gains were ineffective in advancing the patient in her treatment plan. Attempts to regulate voiding and bowel function were similarly unsuccessful. However, she was successful in learning one-handed activities of daily living and the operation of a single-arm-drive wheelchair, which was introduced late in her stay after other psychiatric and physical medicine interventions proved unsuccessful. The patient was closely followed by two psychiatrists during her rehabilitation unit stay. An attempt to hypnotize her was unsuccessful except with several of the residents observing the induction. A detailed social history was taken; the patient reported that her father had been a physician and her mother a registered nurse, but

that both parents and her only child were deceased. She reported that she had a brother, but that she had lost contact with him. She stated that she had recently been in training to be a substanceabuse counselor, and gave a history of remote personal alcohol abuse. She reported recently moving to the city and staying with friends, though she rarely had visitors. Most of the history could not be confirmed. Late in her stay, investigation showed that her local address, staying with, Records from for pulmonary

given by the patient as that of a friend she was to be a shelter for abused and homeless women. the hospital where she claimed to have been treated embolism showed only two emergency room visits

for severe headaches, treated with narcotic analgesics. She was discharged to a local nursing home, where her symptoms progressed to complete quadriplegia and blindness. She was admitted to another hospital, where she was again exhaustively

worked-up for physical pathology, and discharged to the nursing home. About 1 month later, after her initial claim for social security disability was denied, she disguised herself and walked out of the nursing home, and was lost to follow-up.

DISCUSSION of chronic factitious disorder with physical symptoms (Munchausen syndrome) is unknown, but the disorder is probably rare.9 A survey of admissions to a teaching hospital over a lo-year period found a total of 41 Fes of factitious disorder as a primary diagnosis, of whom only three were possibly Munchausen syndrome.‘o The incidence of the chronic pain and psychological variants of Munchausen syndrome may be higher; a survey of 775 admissions to a psychiatric hospital showed four patients admitted with this diagnosis,” and a survey of 2,860 admissions to a chronic pain program showed four patients with chronic factitious pain syndrome.” Starting with Asher’s classic article,13 the neurological variant of Munchausen Syndrome has been frequently reported.14-16Many of these patients may seem from a functional standpoint to be good candidates for rehabilitation, The true incidence

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Table 1: Diagnostic Criteria for 301.51 Factitious Disorder With Physical Symptoms Intentional production or feigning of physical (but not psychological) symptoms. A psychological need to assume the sick role, as evidenced by the absence of external incentives for the behavior, such as economic gain, better care, or physical well-being. Occurrence not exclusively during the course of another Axis I disorder, such as schizophrenia. (Adapted from DSM III-R.9)

although there is only a single Munchausen case report in the rehabilitation literature, and this patient does not seem to have been admitted for inpatient rehabilitation.17 It is likely that most of these patients are never referred for rehabilitative interventions. The presence of functional symptoms does not rule out underlying physical pathology, and a reasonable set of screening tests is an essential first step in the management of suspected functional disorders. However, the clinician should be aware that extensive testing cannot exclude organic disease with certainty, and with every added test the likelihood of spuriously positive results increases.18 In the present case, a mildly abnormal VERS resulted in the diagnosis of multiple sclerosis being entertained throughout the patient’s hospitalization, because the development of conversion symptoms following the diagnosis of multiple sclerosis has been described.” Other disorders that are occasionally misdiagnosed as functional include Guillian-Barre syndrome, myasthenia gravis, porphyria, uremia, panhypopituitarism, insulinoma, Addison’s disease, botulism, central cord syndrome, and carbon monoxide and heavy metals poisoning.20 Clinical examination findings in functional paralyses and anesthesias have been described’8*20;however, the finding of a functional neurological deficit does not show whether the deficit is symptomatic of a conversion disorder, or is consciously feigned, as in factitious disorders and malingering. This is usually apparent from the history; however, as the present case indicates, the limited and inaccurate history usually obtained from such patients may prevent accurate diagnosis. Elements suggesting chronic factitious disorder include a history of recent moves and multiple medical procedures, on a background of dramatic, often unbelievable personal history, referred to as ‘ ‘pseudologia fantastica.“’ DSM III-R criteria for factitious disorder with physical symptoms and conversion disorder are listed in tables 1 and 2. Features suggesting malingering are listed in table 3. Proposed changes for DSM IV diagnosis of factitious disorder will combine physical, psychological, and chronic pain variants of factitious disorder under a single heading.21 Additional features that may aid in the identification of Munchausen Syndrome are listed in table 4. The case in this report combined features of all three diagnoses; although she seemed to best fit the category of factitious disorder, she showed an absence of response to painful stimulation and depressive features suggestive of a conversion disorder; on the other hand, her attempt to obtain social security disability compensation was more consistent with malingering. Part of the difficulty in accurately diagnosing patients with

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FACTITIOUS TRIPLEGIA, Berry Table 2: Diagnostic Criteria for 300.11 Conversion Disorder A loss of, or alteration in, physical functioning suggesting a physical disorder. Psychological factors are judged to be etiologically related to the symptom because of a temporal relationship between a psychosocial stressor that is apparently related to a psychological conflict or need and initiation or exacerbation of the symptom. The person is not consciously aware of intentionally producing the symptom. The symptom is not a culturally sanctioned response pattern and cannot, after appropriate investigation, be explained by a known physical disorder. The symptom is not limited to pain or to a disturbance in sexual functioning. Specify: single episode or recurrent. (Adapted from DSM III-R.9)

this disorder is related to whether the factitious syndrome is itself a primary disorder, or is symptomatic of another, underlying psychiatric disorder. A review of case reports22 found that patients seemed to cover a spectrum of underlying disorders, with some cases so disorganized as to be diagnosed as schizophrenic, others with apparent neuroses, and perhaps the largest percentage with personality disorders. The author noted a history of drug addiction or observed drug-seeking behavior in 75% of reports, and suggested that substance abuse might be the most common underlying disorder. However, although our patient displayed considerable drug-seeking behavior early in her hospital course, she did not give up her triplegia once all narcotics and tranquilizers were stopped. No consensus exists as to the appropriate management of patients with Munchausen syndrome, and few cases of successful treatment have been reported. There is no previous report of attempts to treat using a behavior modification protocol similar to those effective with conversion disorders. Because hospitalization and treatment itself is reinforcing for these individuals, and because the elaboration and development of new symptoms is typical, moving in a stepwise program toward greater independence is likely to be ineffective, as it was in this case. The most appropriate management for these individuals may be to obtain early psychiatric consultation to establish the probable diagnosis, to use the psychiatric consultant to aid the team in dealing with the patient’s behavior, and to offer the patient psychiatric follow-up.293Although the value of confrontation is debatable,6X’8Z23*24 reported attempts at Table 3: Features of V65.20 Malingering The essential feature of malingering is intentional production of false or grossly exaggerated physical or psychological symptoms, motivated by external incentives Malingering should be strongly suspected if any combination of the following is noted: Medicolegal context of presentation Marked discrepancy between the person’s claimed stress or disability and the objective findings; Lack of cooperation during the diagnostic evaluation and in complying with the prescribed treatment regimen The presence of antisocial personality disorder (Adapted from DSM III-R?)

Table 4: Associated Features Suggestive of Factitious Disorder Pseudologia fantastica Disruptive behavior and noncompliance with treatments Extensive knowledge of medical terminology and routines Overuse of analgesics for “pain” Evidence of multiple prior surgeries History of extensive travel Few or no visitors Fluctuating clinical course, with new complications and pathology when initial work-up is negative (Adapted from DSM III-R.9)

nonconfrontational management and “face-saving” approaches seem to have accomplished little except for prolonging hospitalizations. In the present case, the patient did not give up her symptoms until finally discharged from the hospital to a nursing home and receiving a negative disability determination from the social security office. CONCLUSIONS Although factitious disorder with physical symptoms is uncommon, it is a disorder in which early diagnosis can save the rehabilitation provider a great deal of time and effort, and spare the health care system the expenditure of costly resources.25326Keys to early diagnosis include identification of functional symptoms on physical examination, careful history including background investigation when indicated, adequate but not excessive search to rule out underlying physical disorders, and early psychiatric consultation to confirm the diagnosis and aid in the patient’s appropriate management. It has been suggested that this disorder is actually fostered by the nature of modem health care delivery, and that the cure for this condition may only lie in modification of the system to make such patients’ behavior less reinforcing.26.27Until that occurs, health care providers will occasionally be forced to deal with the ethica128.2gand medicolega130*3’ dilemmas posed by such patients. References

1. Cramer B, Gershber M, Stem M. Munchausen syndrome: its relationship to malingering, hysteria, and the physician-patient relationship. Arch Gen Psychiatry 1971;24:575-8. 2. Hyler S, Sussman N. Chronic factitious disorder with physical symptoms (the Munchausen syndrome). Psychiatr Clin North Am 1981;4:365-77. 3. Nadelson T. The Munchausen spectrum: borderline character features. Gen Hosp Psychiatry 1979; 1: 11-17. 4. Taylor S, Hyler S. Update on factitious disorders. Int J Psychiatry Med 1993;23:81194. _ 5. Trieschman R, Stolov W, Montgomery A. An approach to the treatment of abnormal ambulation resulting from conversion reaction. Arch Phys Med Rehabil 1970;5 1: 198-206. ” 6. Eisendrath S. Factitious physical disorders: treatment without confrontation. Psychosomatics 1989;30:383-7. 7. Yassa R. Munchausen syndrome: a successfully treated case. Psychosomatics 1978; 19:242-3. 8. Solyom C, Solyom L. A treatment program for functional paraplegia/ Munchausen syndrome. J Behav Ther Exp Psychiatry 1990;21:225-30. 9. Diagnostic and Statistical Manual of Mental Disorders (3rd ed rev), Washington, DC: American Psychiatric Association, 1987. 10. Reich P, Gottfried L. Factitious disorders in a teaching hospital. Ann Intern Med 1983;99:240-7. 11. Bhugra D. Psychiatric Munchausen syndrome. Acta Psychiatr Stand 1988;77:497-503.

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FACTITIOUS TRIPLEGIA, Berry 12. Fishbain D, Goldberg M, Rosomoff R, Rosomoff H. More Munchausen with chronic pain. Clin J Pain 1991;7:237-44. 13. Asher R. Munchausen syndrome. Lancet 1951; 1:339-41. 14. Folger W, Lipton R, Rovner R, Haster D. Neurologic Munchausen syndrome (letter). Neurology 1981;31:638-9. 15. Fraim C, Peters B. Munchausen syndrome (letter). Ann Neurol 1979;6:138. 16. Biver F, Delveune V, Hirsch D, .Lotstra F. Factitious hemiplegia and Munchausen syndrome. Acta Neurol Belg 1992;92:289-95. 17. Lazar R. Munchausen syndrome presenting as acute spinal cord injury. Arch Phys Med Rehabil 1986;67:568-9. 18. PurcellT.Thesomaticpatient.EmergMedClinNorthAm 1991;9:13759. 19. Caplan L, Nadelson T. Multiple sclerosis and hysteria: lessons learned from their association. JAMA 1980;243:2418-21. 20. Tandberg D. Diagnosis of nonorganic coma, seizures, weakness and numbness. Resident and Staff Physician 1982;28:62-8. 21. American Psychiatric Association Task Force on DSM-IV. DSM-IV Options Book: Work in Progress. Washington, DC: American Psychiatric Association.

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22. Mendel J. Munchausen syndrome: a syndrome of drug dependence. Compr Psychiatry 1974; 15:69-72. 23. Ireland P, Sapira J, Templeton B. Munchausen syndrome: review and report of an additional case. Am J Med 1967;43:579-92. 24. Wedel K. A therapeutic confrontation approach to treating patients with factitious illness. Sot Work 1971; 16:69-73. 25. Powell R, Boast N. The million dollar man: resource implications for chronic Munchausen syndrome. Br J Psychiatry 1993; 162:847-8. 26. Black W. Resource implications of Munchausen syndrome. Br J Psychiatry 1993; 162:847-8. 27. Higgins P. Temporary Munchausen syndrome. Br J Psychiatry 1990;157:613-6. 28. Sadler J. Ethical and management considerations in factitious disorder: one and the same. Gen Hosp Psychiatry 1987;9:31-6. 29. Ford C, Zaner R. Response to the article “Ethical and management considerations in factitious disorder: one and the same” by John Z. Sadler. Gen Hosp Psychiatry 1987;9:37-9. 30. Houck C. Medicolegal aspects of factitious disorder. Psychiatr Med 1992; 10:105-16. 31. Lipsitt D. The factitious patient who sues (letter). Am J Psychiatry 1986; 143:1482.